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Stereotypic Movement Disorders

Katherine Mackenzie

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DOI: https://doi.org/10.1016/j.spen.2017.12.004
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To appear in: Seminars in Pediatric Neurology
Cite this article as: Katherine Mackenzie, Stereotypic Movement Disorders,
Seminars in Pediatric Neurology,doi:10.1016/j.spen.2017.12.004
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Stereotypic Movement Disorders

Katherine Mackenzie, MD

Division of Child Neurology, Department of Neurology, Stanford University Palo Alto,

California

Address reprint requests to:

Katherine Mackenzie, MD, Division of Child Neurology, 750 Welch Road Suite 317,

Palo Alto, CA 94304. [email protected]

Disclosure of interests: The authors have no commercial, proprietary, or financial interest

in any products or companies described in this article.

Abstract

This review summarizes motor stereotypies in terms of diagnosis, description, prevalence,

pathophysiology and management. Stereotypies are fixed and chronic movements. Stereotypies

begin before 3 years of age and continue into adulthood. Primary motor stereotypies occur in

children of normal intelligence whereas secondary stereotypies occur in the setting of an

additional diagnosis such as Autism Spectrum Disorder (ASD) or other neurologic disorders.

They are highly associated with co-morbidities such as anxiety, obsessive-compulsive

symptoms, inattention, and tics. The pathophysiology of stereotypies involves frontostriatal

overactive dopaminergic pathways, and underactive cholinergic and GABAergic inhibitory

pathways. No genetic markers have been identified despite a clear genetic predisposition.
Behavioral therapy is the principle treatment. Future studies will focus on identifying genetic

markers, and on better understanding the functional and structural neurobiology of these

movements.

Introduction

Stereotypies are described as repetitive and purposeless movements that occur in a specific

pattern and are distractible.1 These movements tend to occur more at times of increased stress,

anxiety, excitement, focused concentration, or boredom.1, 2 Simple motor stereotypies (aka

physiologic stereotypies or common behaviors) occur commonly in both children and adults.

These include leg shaking, hair twirling, and nail biting. Complex motor stereotypies are more

complex movements such as hand flapping, finger/arm wiggling, mouth opening, orofacial

movements, and body rocking.1 Alterations in breathing patterns or vocalizations may also

accompany the movements. Both types of stereotypies, simple and complex, are also divided

into two categories as either primary or secondary, depending on the presence of an additional

neurologic or psychiatric diagnosis.3 Table 1 outlines common examples of stereotypies.

Description and Prevalence

Children with stereotypies often report that these movements are a pleasant experience, and

may feel frustrated when parents and teachers interrupt them.4 One child described her

movements as follows, “it just feels right…sometimes I hold my hands by my side to stop the

movements but it doesn’t feel nice”.5 One theory is that the movements are a way of physically

expressing or dealing with excitement, or can be a way of coping with boredom (i.e. in both

under- and overstimulating environments).6 Older children and adults may learn to suppress

movements, or attempt to mask stereotypies with other purposeful movements, when in front of

others. A subset of children with stereotypies report episodes of intense imagery during their
movements, such as imagining that they are part of a video game or cartoon. 5 Self-injurious

behaviors are generally also considered to be complex motor stereotypies on a more severe

level, as they rarely occur in children without other motor stereotypies.7

Primary simple motor stereotypies are reported to occur in roughly 20-70% of typically

developing children, whereas the prevalence of complex motor stereotypies is reported at

roughly 3-4%.5, 8, 9, 10, 11 However, in children with developmental disabilities, the prevalence of

stereotypies is reported to be high at 61%, and is even higher in children with ASD at 88%.12

Complex motor stereotypies typically begin before 3 years of age, with peak onset around 12

months, and persist throughout adolescence and adulthood. 2,13 One study found that 80% of

children with stereotypies developed their movements before 24 months of age, with only 8%

presenting after 36 months of age. 2

Stereotypies also appear to be persistent throughout life. The largest longitudinal study of

children/adolescents with primary complex motor stereotypies (ages between 9-20 years old)

found that 98% of participants continued to have stereotypies but roughly 80% reported that

they were improved/more manageable with older age.13 An earlier 2008 study by this group

reported that of the 98 children surveyed, only 6% experienced resolution of their stereotypy.

Resolution was more likely if the child’s primary stereotypy was head nodding (38% of head

nodding resolved).2

Primary Complex Motor Stereotypies occur in people without an underlying neurodevelopmental

disorder and with normal intelligence. The term “Secondary Stereotypies” refers to stereotyped

movements that occur in the setting of an additional diagnosis such as ASD, intellectual

disability delay, sensory delays, or genetic disorders such as Rett syndrome, Lesch-Nyhan,
Cornelia de Lange, Angelman syndrome, and Fragile X syndrome.14,15 There is also a known

association of adult onset disorders and stereotypies, including Frontotemporal Dementia and

Schizophrenia.16 Less common causes of these movements include tumors, infections, or

autoimmune disorders and drugs such neuroleptics, amphetamines, or cocaine.17

Children with sensory deficits such as blindness have a high incidence of stereotypies. Part of

the underlying theory that stereotypies can be a response to an “under-stimulating environment”

is based on this observation. For example, one study reported an incidence of stereotypy in

blind children of roughly 70%, and included the following in order of frequency: body rocking

(30%), repetitive handling of objects (31%), hand and finger movements (28%), and eye

pressing and eye poking (31%). They also noticed an increase in stereotypies with decreased

environmental stimulation and increased restriction.18 Another study found that 100% of the 85

blind children surveyed performed stereotyped movements (eye poking, body rocking, etc.) that

increased during times of “monotony, arousal, demand, and during feeding or eating”. 19

Assessment of children with stereotypies

There are 3 main care-giver based rating scale questionnaires that can be helpful in evaluating

stereotypies in children that are based on care-giver reporting: 1) Motor Stereotypy Severity

Scale/SSS (see Table 2 for description20) broken down into three components: SSS Motor

(number, frequency, intensity, interference), SS Impairment to characterize global impairment,

and a Linear Analog Scale 21,22 2) Repetitive Behavior Scale (see Table 3 for description23,24)

with six subscales to assess for stereotyped behavior, self-injurious behavior, compulsive

behavior, ritualistic behavior, sameness behavior, and restricted behavior 23,24,,25,26 and 3)

Behavior Problems Inventory/BPI (see Table 4 for description27,28) with three subscales for

self-injurious behavior, stereotyped behavior, and aggressive/destructive behavior.

In terms of clinical diagnosis, Table 5 lists the DSM-V diagnostic criteria. 29 Included in the

DSM-V criteria is the finding that the movement “interferes with social, academic, or other

activities and may result in self-injury”. As such, it is important for clinicians to discuss with

parents and patients whether these movements are causing interference in daily activities. In

addition, evaluation for other neurologic/psychiatric comorbidities (Table 6), that can be

associated with stereotypies, is an important part of the evaluation process. Many of these

comorbidities are also understood to involve dysfunction of the fronto-striatal circuits in the

brain. One large study identified comorbidities in roughly 90% of children surveyed based on

parental/self-reports of symptoms, and checklists for attention-deficit/hyperactivity disorder

(ADHD), Obsessive Compulsive Syndrome/Disorder (OCS/OCD) and anxiety as follows: ADHD

(63%), tics (22%), OCD (35%), and anxiety (73%).13 Another study of children with the

diagnosis of primary complex motor stereotypy found that 1/3 had symptoms of Developmental

Coordination Disorder, defined as learning and execution of coordinated motor skills below

expected level for age.3,29 The parents of these children also reported more symptoms of

inattention and executive dysfunction.3

Differential Diagnosis:

A frequent question from referring physicians to pediatric neurology clinics is whether these

movements could represent a seizure or a motor tic. Through a careful history, review of home

videos, and observation in the clinical setting, these diagnoses can generally be distinguished

from one another as described.

Parents and teachers often raise concerns that stereotypies could be seizures. The semiology

of the movements typically points towards the appropriate diagnosis. Distinguishing features of

stereotypies include situationally-specific triggers for movements such as times of excitement or

boredom. Conversely, seizures are not typically triggered by a change in emotions, attention, or
cognitive tasks. The ability to redirect a child or interrupt the movements is also a key

distinguishing feature of stereotypies, as seizures are generally not suppressible. Frequency

may also be helpful in that stereotypies can happen many times a day, whereas most seizures

are less frequent and tend to occur in discreet episodes with a relatively clear on and off timing.

Motor tics are frequently confused with stereotypes. Motor tics involve a preceding undesired

sensation that is relieved once the movement is completed. This phenomenon is not described

by people with stereotypies. As mentioned above, stereotypies often provide a pleasing

experience for children when performed. It is not uncommon for a child with stereotypies to

express frustration or irritation when interrupted by others.

Tics also tend to involve discreet muscle groups in a specific repetitive pattern (blink and then

head nod to the right, blink and then head nod to the right), whereas stereotypies can be more

variable from movement to movement (hand rotational movements change to arm flapping with

body rocking). Additional clues to help distinguish between these two types of movements

include age of onset. Tics tend to present in children older than 3 years of age, whereas the

average age of presentation for stereotypies almost always begin before 3 years of age. Tics

often change over time, whereas stereotypies tend to be the same throughout a person’s

lifetime. Both tics and stereotypies are suppressible, at least temporarily, but tics are usually

internally suppressed whereas stereotypies are externally suppressed (i.e. by the parent or

teacher). Both movements improve with specific behavioral therapy but are only indicated if the

movements are impairing the child. Medications can be helpful in modulating tics, whereas

medication trial results have been disappointing in reducing stereotypies. Primary differences

between motor tics and stereotypes are described in Table 7.

Pathophysiology

Stereotypies are associated with dysfunction of the prefronto-corticobasal ganglia circuits, or

cortico-striatal-thalamo-cortical pathways.15,30 They are clearly linked to dopaminergic

overstimulation given that they are a side effect from dopaminergic drugs such as

amphetamine, cocaine, and levodopa.31 Similarly, lesioning dopaminergic input to striatal

neurons prevents stimulant-induced stereotopy.32 Inhibitory cholinergic interneurons in these

regions also play a role in modulating and suppressing stereotypies.33 Y-aminobutyric acid

(GABA) also appears to play a role. A recent imaging study also showed a correlation of

decreased striatal and anterior cingulate cortex GABA levels with increased complex motor

stereotypies in children.34

Although stereotypies can be modified, interrupted, or partially suppressed, one

electrophysiologic study distinguished stereotypies from voluntary movements based on EEG

recordings of movement-related cortical potentials (MRCPs). This study demonstrated that

MRCPS seen in the prefrontal cortex prior to voluntary movements were absent prior to

stereotypies.35

Structural brain imaging studies have reported various differences in the brains of people with

stereotypies as well. For example, one study of adult males reported decreased frontal white

matter and caudate nuclei volume in participants with stereotypies.36 Another study reported

smaller sized putamen.37 There are also some case reports of focal lesions resulting in

stereotypies. One woman with a unilateral lentiform nucleus stroke developed parkinsonism and

stereotypies that improved with pimozide.38 Another study reported stereotypies and autistic

behavior following a unilateral putaminal infarct.39

Children with complex motor stereotypies are more likely to have a family member with

stereotypies. Reports of a positive family history have ranged between 17-39 %, the majority of

whom are first-degree relatives.2, 13, 40, 41 To date, no specific genes have been identified in

stereotypies but current research is focused on specific genes with known links to Autism

Spectrum Disorder in individuals with primary complex motor stereotypies 22, 42

Management

The majority of stereotypies does not cause significant physical or emotional distress, and often

do not require intervention. For stereotypies that are bothersome, habit reversal therapy can be

successful in reducing their severity and frequency.43 In the classroom or home setting,

response interruption and redirection when performed regularly and consistently can also be

effective.44 A recent program aimed at DVD-based parent-delivered behavioral therapy showed

reduction in stereotypies by 15-24%.45

For children with secondary stereotypies and self-injurious behavior, pharmacotherapy may be

indicated. Psychoactive drugs such as clomipramine, risperidone, and fluoxetine have

demonstrated efficacy in reducing stereotypy for children with Autism. 46,47,48 However, there are

no current blinded control studies on pharmacologic interventions in children with primary

complex motor stereotypy. i.e. without an underlying neurodevelopmental disorder.49 A

longitudinal study of children with primary complex motor stereotypy reviewed medications

prescribed for other comorbidities such as ADHD or OCD, and found that no difference in

stereotypy severity was reported by patients while taking medications that included

methylphenidate, atomoxetine, amphetamine mixed salts, lisdexamfetamine,

dexmethylphenidate, guanfacine, fluoxetine, lamotrigine, topiramate and excitalopram.13

Conclusion

Motor stereotypies are relatively common in childhood and can occur with or without additional

comorbidities, and/or in the setting of additional neurodevelopmental disorders. Complex motor

stereotypies tend to present in early childhood and often do not change over a person’s lifetime.

Current understanding of pathophysiology suggests involvement in dopaminergic pathways, and

to some extent GABAergic and cholinergic pathways, in the prefronto-corticobasal ganglia

circuits and cortico-striatal-thalamo-cortical circuits. To date, no clear genetic mutations have

yet been identified despite a known increased incidence of stereotypies among family members

with these movements. Most primary stereotypies do not require treatment, but habit reversal

training can be effective for children who desire help in reducing their movements. However, for

children with secondary stereotypies (i.e. other neurodevelopmental disorders) and self-injurious

behaviors, medication options have been found to be effective.

Table 1.

Examples of Stereotypies

Simple Motor Stereotypy

Leg shaking

Thumb sucking

Nail biting

Teeth clenching/grinding

Complex Motor Stereotypy

 Hand flapping

Hand/arm waving

Rotating or opening and closing the hands

Finger wiggling

Mouth opening

Orofacial movements

Table 2.
Motor Stereotypy Severity Scale/SSS. 20
Number 0 None
1 Single stereotypy
2 2-5 discrete stereotypies
3 >5 discrete stereotypies

Frequency 0 Never
1 Rarely Not daily
2 Occasionally Daily, but infrequent
3 Frequent Daily, multiple times per day
4 Very Frequent Virtually every hour
5 Always Few if any, stereotypy-free intervals

Intensity 0 Absent
1 Minimal Minimally forceful compared to voluntary actions and not visible
2 Mild Not more forceful than comparable voluntary actions and not usually noticed
3 Moderate More forceful than comparable voluntary actions and call attention to
individual
4 Marked More forceful than comparable voluntary actions, exaggerated, and call
attention
5 Severe Extremely forceful and exaggerated, call attention, may cause physical
injury

Global 0 None
Impairment 10 Minimal Associated with subtle difficulties in self-esteem, family, school, or social
acceptance
Rating 20 Mild Associated with minor problems in self-esteem, family, school, or social
acceptance
30 Moderate Associated with clear problems in self-esteem, family, school, or social
acceptance
40 Marked Associated with major difficulties in self-esteem, family, school, or social
acceptance
50 Severe Associated with extreme difficulties in self-esteem, family, and severely
restricted life
because of social stigma and school avoidance
Table 3.
Repetitive Behavior Scale – Revised Subscales (43 item questionnaire for parents). 23, 24

Stereotyped Behavior
Self-injurious Behavior
Compulsive Behavior
Ritualistic Behavior
Sameness Behavior
Restricted Behavior

Table 4.
Behavior Problems Inventory 27, 28
SELF INJURIOUS BEHAVIOR
1. Self-biting (so hard that a tooth print can be seen for some time; bloodshot or breaking of skin may occur)
2. Hitting head with hand or other body part (e.g., face slapping, knee against forehead) or with/against objects (e.g., slamming
against a wall, knocking head with a toy)
3. Hitting body (except for the head) with own hand or with any other body part (e.g., kicking self, slapping arms or thighs), or
with/against objects (e.g., hitting legs with a stick, boxing the wall)
4. Self-scratching (so hard that reddening of the skin becomes visible; breaking of the skin may also occur)
5. Vomiting and rumination (deliberate regurgitation of swallowed food with rumination)
6. Self-pinching (so hard that reddening of the skin becomes visible; breaking of the skin may occur)
7. Pica: Mouthing or swallowing of objects which should not be mouthed or swallowed for health or hygiene reasons (non-food items
such as feces, grass, paper, garbage, hair)
8. Inserting objects in body openings (in nose, ears, or anus, etc.)
9. Pulling finger or toe nails
10. Inserting fingers in body openings (e.g., eye poking, finger in anus)
11. Air swallowing resulting in extended abdomen
12. Hair pulling (tearing out patches of hair)
13. Extreme drinking (e.g., more than 3 liters per day)
14. Teeth grinding (evidence of ground teeth)
15. Other: ……………………………………………….

STEREOTYPED BEHAVIOR
16. Rocking back and forth
17. Sniffing objects
18. Spinning own body
19. Waving or shaking arms
20. Rolling head
21. Whirling, turning around on spot
22. Engaging in repetitive body movements
23. Pacing
24. Twirling things
25. Having repetitive hand movements
26. Yelling and screaming
27. Sniffing own body
28. Bouncing around
29. Spinning objects
30 Having bursts of running around
31. Engaging in complex hand and finger movements
32. Manipulating objects repeatedly
33. Exhibiting sustained finger movements
34. Rubbing self
35. Gazing at hands or objects
36. Maintaining bizarre body postures
37. Clapping hands
38. Grimacing
39. Waving hands
40. Other ....................................................................

AGGRESSIVE/DESTRUCTIVE BEHAVIOR
41. Hitting others
42. Kicking others
43. Pushing others
44. Biting others
45. Grabbing and pulling others
46. Scratching others
47. Pinching others
48. Spitting on others
49. Being verbally abusive with others
50. Destroying things (e.g., rips clothes, throws chairs, smashes tables)
51. Being mean or cruel (e.g., grabbing toys or food from others, bullying others)
52. Other: ….............................................
Parents are asked to quantify Frequency (0=Never, 1=Monthly, 2= Weekly, Daily=3, Hourly=4)
and Severity (Mild=1, Moderate=2, Severe=3)

Table 5. DSM-V Diagnostic Criteria of Stereotypic Movement Disorder 14

Stereotypic Movement Disorder ICD-10 F98.4

A. Repetitive, seemingly driven, and apparently purposeless motor behavior (e.g., hand

shaking or waving, body rocking, head banging, self-biting, hitting own body).

B. The repetitive motor behavior interferes with social, academic, or other activities and

may result in self-injury.

C. Onset is in the early developmental period.

D. The repetitive motor behavior is not attributable to the physiological effects of a

substance or neurological condition and is not better explained by another

neurodevelopmental or mental disorder (e.g., trichotillomania [hair-pulling disorder],

obsessive-compulsive disorder).
Table 6.

Comorbid Diagnoses Associated with Primary Motor Stereotypies

ADHD

Anxiety

OCD

Tics

Developmental Coordination Disorder

Learning Disorders
Table 7.

Differentiating Tics from Motor Stereotypies

Tics Stereotypy

Age of onset >3 yo < 3yo

Duration Seconds Seconds-minutes

Premonitory urge Yes No

Fixed over time No Yes

Enjoyable No Yes

Suppression Self-imposed Imposed by others

Movement Description Discreet muscle groups in Similar muscle groups in a

the same pattern similar pattern

Common Muscles Facial muscles, neck, proximal Upper extremities, trunk,

upper extremities head, mouth

Behavior Therapy Comprehensive Behavioral Habit Reversal Training

Intervention for Tics (CBIT) Response Interruption

and Redirection

Medication Alpha-2-agonists Not typically helpful

Dopamine Antagonists

Serotonin Reuptake Inhibitors

Monoamine Depleters
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